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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 11  |  Issue : 2  |  Page : 33-35

Pleomorphic adenoma of the trachea as a finding during coronavirus disease 2019 workup and diagnosis


1 Department of ENT and Surgery, IMSS UMAE 189 CMN 14, Veracruz, Mexico
2 Departments of ENT and Surgery, IMSS UMAE 189 CMN 14; Department of Surgery, Hospital de Alta Especialidad de Veracruz, Sesver, Veracruz, Mexico
3 Department of Surgery, Hospital de Alta Especialidad de Veracruz, Sesver; Department of Research, School of Medicine, Universidad Cristobal Colon, Veracruz, Mexico

Date of Submission15-Jul-2021
Date of Acceptance19-Dec-2021
Date of Web Publication12-Apr-2022

Correspondence Address:
Enrique Sanchez-Valdivieso
Av. 20 De Noviembre Sn, Cp 91700, Veracruz
Mexico
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jlv.jlv_2_21

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   Abstract 


Very small number of minor salivary glands is seen around the larynx and trachea. A tumor originating from the salivary glands is infrequently seen in the respiratory tract. Pleomorphic adenoma (PA) is a benign tumor that can be seen on the head and neck area, most frequently involving major salivary glands. PA originating from the tracheobronchial tree is very rare. We describe here one of such cases, detected during the workup of a health-care worker that became infected with coronavirus disease 2019. Clinicians should be aware of benign tumors arising from the tissues of the central airways and avoid late diagnosis.

Keywords: Minor salivary gland, pleomorphic adenoma, trachea


How to cite this article:
Ventura-Morales B, Castillo Marquez Y, Gomez-Calzada JG, Sanchez-Valdivieso E. Pleomorphic adenoma of the trachea as a finding during coronavirus disease 2019 workup and diagnosis. J Laryngol Voice 2021;11:33-5

How to cite this URL:
Ventura-Morales B, Castillo Marquez Y, Gomez-Calzada JG, Sanchez-Valdivieso E. Pleomorphic adenoma of the trachea as a finding during coronavirus disease 2019 workup and diagnosis. J Laryngol Voice [serial online] 2021 [cited 2022 Dec 5];11:33-5. Available from: https://www.laryngologyandvoice.org/text.asp?2021/11/2/33/342861




   Introduction Top


There are around 1000 minor salivary glands in the human body, which vary in size from 1 to 5 mm embedded in the submucosa of the aero-digestive tract and the auditory tube,[1],[2] more concentrated in the region of the head and neck, scattered throughout the oral cavity, sinonasal cavities, and oropharynx. A small number are also seen around the larynx (supra-glottis) and trachea; any of these glands may become a source of disease.[1]

Very rarely, a benign tumor of the salivary glands is seen in the respiratory tract. Tumors in this region grow slowly, causing symptoms mainly dependent on the size and location of the tumor. Tumors located in the trachea cause the symptoms of obstruction; patients may present with chronic cough or dyspnea, and recurrent respiratory infections can also be seen. In patients with tracheal tumors, a late diagnosis is to be expected, since it may resemble chronic obstructive pulmonary disease or asthma.

Pleomorphic adenoma (PA) is a type of benign salivary gland tumor that can be seen on the palate, lip, septum and nasopharynx, orbital area, cheek, and external auditory canal.[3] This tumor is composed of the epithelial tissue mixed with mucoid, myxoid, and chondroid tissues. It consists of myoepithelial cells mixed with neoplastic ducts and stroma. PA rarely occurs in the trachea.[4] PA originating from the tracheobronchial tree represents between 2% and 9% of all PA cases.[5] We describe here one of such cases.


   Case Report Top


A 27-year-old male, born in Mexico City, resident of the City of Veracruz, a family physician, who was referred to our hospital due to the computed tomography (CT) finding of a polyp located in the trachea during a recent diagnosis of coronavirus disease 2019. There was no history of weight loss. The patient was previously diagnosed with asthma at the age of 18 years. There was no history of smoking or alcoholism; refers to a diabetic mother and a maternal grandfather with prostate cancer.

On physical examination, there was no stridor in the neck or lymphadenopathy. The laboratory reported hemoglobin 16.3 g/dl, hematocrit 46.9%, platelets 213,000, leukocytes 5.86 × 103, neutrophils 58.5%, lymphocytes 34%, monocytes 6.1%, eosinophils 0.9%, basophils 0.3%, blood glucose 100 mg/dl, urea 24.3 mg/dl, and creatinine 0.7 mg/dl.

Laryngoscopy was performed, and a smooth and vasodilated mass was visualized. CT virtual bronchoscopy showed a well-defined soft-tissue density lesion of 2.0 cm × 2.0 cm in the upper trachea, located 2 cm below the glottis [Figure 1]a and [Figure 1]b. Fiber-optic bronchoscopy revealed a subglottic lesion dependent on the posterior wall, broad base, free edges, and pink coloration. Fifty percentage of the tracheal lumen was obstructed. The biopsy report was neurofibroma.
Figure 1: (a) Chest computed tomography revealed an endotracheal mass. (b) Laryngoscopy showed a polypoid neoplasm with branching blood vessels on the surface, located below the vocal cords

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The procedure was performed under general anesthesia. A transverse incision was made in the midline of the neck along with a division of the thyroid. An additional 5 cm dissection was performed distal to the tumor site. The dissection was meticulously performed to avoid damage to the recurrent laryngeal nerves. A section of the trachea was made at the site of the tumor. Longitudinal tracheofissure and exposure of the posterior trachea were performed, achieving visualization of a 2 cm × 2 cm lesion adherent to cartilage, of solid appearance, stony consistency, attached to the mucosa immediately distal to the cricoid cartilage, which was completely excised. Next, both ends of the trachea were anastomosed. Tracheal reconstruction was performed with separate stitches of 2-0 vicryl™; a Shiley tracheostomy cannula was positioned. On the 10th postoperative day, we confirmed that the anastomosis site was healing and that the upper tracheal lumen remained at approximately 80% patency. Two months after the operation, the area of the tracheoplasty was visually healthy. A follow-up CT scan of the neck revealed a wide tracheal lumen.

The result of the pathological examination was a pleomorphic adenoma. Histopathological analysis revealed a tumor composed of myxoid and mesenchymal epithelial elements and was characterized by the presence of ductal structures that appeared to contain a double layer of cells in a mucoid or hyaline stroma. No abnormal mitoses were observed [Figure 2].
Figure 2: Medium-magnification photomicrograph of a neoplastic lesion showing small, regular and symmetrical ducts that alternate with myoepithelial cells of clear cytoplasm, located in a basal position, which appear loose and in small groups among a myxoid stroma, without atypia (H and E; ×10)

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   Discussion Top


The median age of patients with PA of the trachea has been reported as 48 years (range 8–83 years).[6] No gender predominance has been reported. The most frequent symptoms were cough, stridor, dyspnea, and wheezing, depending on the site and degree of airway obstruction. It can be misdiagnosed as asthma.[6] Because patients initially present with nonalarming symptoms that mimic asthma,[5] these tumors fall into a category of late diagnosis.

Tracheal tumors are difficult to identify on chest X-rays. Endotracheal lesions can be missed on plain radiographs, but are easily detected with CT scans and virtual bronchoscopy.[7] Depending on the site and size, tumor may appear as a well-defined, multilobulated mass on CT and magnetic resonance imaging (MRI). Calcification may be noted in CT images. On MRI, they are T2 hyperintense tumors with nodular enhancement and often with a T2 dark rim.[8]

In addition, in the presence of resistant cough and dyspnea, the tracheobronchial system should be evaluated with flexible bronchoscopy. Therefore, chest CT and bronchoscopy play a fundamental role in early diagnosis.

To identify the nature of the lesion, bronchoscopy can be used to complete this by biopsy. Cytology and biopsy typically show a tumor of mixed epithelial cells and mesenchymal elements with no pseudocapsule.[9] Histologically, PA is also known as a “mixed tumor,” describing its pleomorphic appearance rather than its dual origin of epithelial and mesenchymal components. The stroma can be mucoid, myxoid, cartilaginous, or hyaline.

Given the rarity of tracheal PA, its biological behavior and clinical course have not been well described. Therefore, standards for the management have not been established, but it is clear that the primary goal is to remove the injury and restore airway patency. Surgical resection and airway anastomosis have traditionally been applied in many studies. Bronchoscopic excision or excision by tracheostomy can also be used to treat these patients.

Similarly, there is no clearly recommended follow-up period or interval, of which the longest follow-up period is 5 years without recurrence.[7] One case of tracheal PA recurred 10 years after resection with end-to-end anastomosis.[10] Therefore, long-term follow-up is essential for patients.

In conclusion, PA rarely presents as a tracheal mass. To avoid late diagnosis, clinicians should be aware that benign tumors arising from the tissues of the central airways could mimic asthmatic symptoms or recurrent airway infections.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Valstar MH, de Bakker BS, Steenbakkers RJ, de Jong KH, Smit LA, Klein NT, et al. The tubarial salivary glands: A potential new organ at risk for radiotherapy. Radiother Oncol 2020;20:30809-14.  Back to cited text no. 1
    
2.
Holsinger F, Bui D. Anatomy, function and evaluation of the salivary glands. In: Myers E, Ferris R, editors. Salivary Gland Disorders. Berlin, Heidelberg: Springer-Verlag; 2007. p. 1-16.  Back to cited text no. 2
    
3.
Kuo YL, Tu TY, Chang CF, Li WY, Chang SY, Shiao AS, et al. Extra-major salivary gland pleomorphic adenoma of the head and neck: A 10-year experience and review of the literature. Eur Arch Otorhinolaryngol 2011;268:1035-40.  Back to cited text no. 3
    
4.
Gaissert HA, Mark EJ. Tracheobronchial gland tumors. Cancer Control 2006;13:286-94.  Back to cited text no. 4
    
5.
Fitchett J, Luckraz H, Gibbs A, O'Keefe P. A rare case of primary pleomorphic adenoma in main bronchus. Ann Thorac Surg 2008;86:1025-6.  Back to cited text no. 5
    
6.
Takahashi M, Yorozuya T, Miyasaka Y, Kodama K, Yoshikawa T, Taya T, et al. A case of tracheal pleomorphic adenoma misdiagnosed as asthma. Oxf Med Case Reports 2019;2019:omz111.  Back to cited text no. 6
    
7.
Finkelstein SE, Schrump DS, Nguyen DM, Hewitt SM, Kunst TF, Summers RM. Comparative evaluation of super high-resolution CT scan and virtual bronchoscopy for the detection of tracheobronchial malignancies. Chest 2003;124:1834-40.  Back to cited text no. 7
    
8.
Ginat DT. Imaging of benign neoplastic and nonneoplastic salivary gland tumors. Neuroimaging Clin N Am 2018;28:159-69.  Back to cited text no. 8
    
9.
Kajiyama A, Edo H, Inoue N, Yokouchi Y, Gomi T. Magnetic resonance imaging and histopathology in a case of pleomorphic adenoma of a minor salivary gland in the nasal cavity. Am J Case Rep 2019;20:679-84.  Back to cited text no. 9
    
10.
Karamustafaoglu YA, Yanık F, Yoruk Y. Palliative treatment of recurrent tracheal pleomorphic adenoma 10 years after segmental resection using the endobronchial shaver. Clin Respir J 2020;14:495-7.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2]



 

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